An overview of methodological approaches in systematic reviews

Abstract Aim The aim of this overview is to identify and collate evidence from existing published systematic review (SR) articles evaluating various methodological approaches used at each stage of an SR. Methods The search was conducted in five electronic databases from inception to November 2020 and updated in February 2022: MEDLINE, Embase, Web of Science Core Collection, Cochrane Database of Systematic Reviews, and APA PsycINFO. Title and abstract screening were performed in two stages by one reviewer, supported by a second reviewer. Full‐text screening, data extraction, and quality appraisal were performed by two reviewers independently. The quality of the included SRs was assessed using the AMSTAR 2 checklist. Results The search retrieved 41,556 unique citations, of which 9 SRs were deemed eligible for inclusion in final synthesis. Included SRs evaluated 24 unique methodological approaches used for defining the review scope and eligibility, literature search, screening, data extraction, and quality appraisal in the SR process. Limited evidence supports the following (a) searching multiple resources (electronic databases, handsearching, and reference lists) to identify relevant literature; (b) excluding non‐English, gray, and unpublished literature, and (c) use of text‐mining approaches during title and abstract screening. Conclusion The overview identified limited SR‐level evidence on various methodological approaches currently employed during five of the seven fundamental steps in the SR process, as well as some methodological modifications currently used in expedited SRs. Overall, findings of this overview highlight the dearth of published SRs focused on SR methodologies and this warrants future work in this area.


INTRODUCTION
Evidence synthesis is a prerequisite for knowledge translation. 1 A well conducted systematic review (SR), often in conjunction with metaanalyses (MA) when appropriate, is considered the "gold standard" of methods for synthesizing evidence related to a topic of interest. 2 The central strength of an SR is the transparency of the methods used to systematically search, appraise, and synthesize the available evidence. 3 Several guidelines, developed by various organizations, are available for the conduct of an SR; 4-7 among these, Cochrane is considered a pioneer in developing rigorous and highly structured methodology for the conduct of SRs. 8 The guidelines developed by these organizations outline seven fundamental steps required in SR process: defining the scope of the review and eligibility criteria, literature searching and retrieval, selecting eligible studies, extracting relevant data, assessing risk of bias (RoB) in included studies, synthesizing results, and assessing certainty of evidence (CoE) and presenting findings. [4][5][6][7] The methodological rigor involved in an SR can require a significant amount of time and resource, which may not always be available. 9 As a result, there has been a proliferation of modifications made to the traditional SR process, such as refining, shortening, bypassing, or omitting one or more steps, 10,11 for example, limits on the number and type of databases searched, limits on publication date, language, and types of studies included, and limiting to one reviewer for screening and selection of studies, as opposed to two or more reviewers. 10,11 These methodological modifications are made to accommodate the needs of and resource constraints of the reviewers and stakeholders (e.g., organizations, policymakers, health care professionals, and other knowledge users). While such modifications are considered time and resource efficient, they may introduce bias in the review process reducing their usefulness. 5 Substantial research has been conducted examining various approaches used in the standardized SR methodology and their impact on the validity of SR results. There are a number of published reviews examining the approaches or modifications corresponding to single 12,13 or multiple steps 14 involved in an SR. However, there is yet to be a comprehensive summary of the SR-level evidence for all the seven fundamental steps in an SR. Such a holistic evidence synthesis will provide an empirical basis to confirm the validity of current accepted practices in the conduct of SRs. Furthermore, sometimes there is a balance that needs to be achieved between the resource availability and the need to synthesize the evidence in the best way possible, given the constraints. This evidence base will also inform the choice of modifications to be made to the SR methods, as well as the potential impact of these modifications on the SR results. An overview is considered the choice of approach for summarizing existing evidence on a broad topic, directing the reader to evidence, or highlighting the gaps in evidence, where the evidence is derived exclusively from SRs. 15 Therefore, for this review, an overview approach was used to (a) identify and collate evidence from existing published SR articles evaluating various methodological approaches employed in each of the seven fundamental steps of an SR and (b) highlight both the gaps in the current research and the potential areas for future research on the methods employed in SRs.

METHODS
An a priori protocol was developed for this overview but was not registered with the International Prospective Register of Systematic Reviews (PROSPERO), as the review was primarily methodological in nature and did not meet PROSPERO eligibility criteria for registration. The protocol is available from the corresponding author upon reasonable request. This overview was conducted based on the guidelines for the conduct of overviews as outlined in The Cochrane Handbook. 15 Reporting followed the Preferred Reporting Items for Systematic reviews and Meta-analyses (PRISMA) statement. 3

Eligibility criteria
Only published SRs, with or without associated MA, were included in this overview. We adopted the defining characteristics of SRs from The Cochrane Handbook. 5   Data related to review characteristics, results, key findings, and conclusions were extracted by at least two reviewers independently (PV performed data extraction for all the reviews and duplicate extraction was performed by AP, HC, or MG).

Quality assessment of included reviews
The quality assessment of the included SRs was performed using the AMSTAR 2 (A MeaSurement Tool to Assess systematic Reviews). The tool consists of a 16-item checklist addressing critical and noncritical domains. 18 For the purpose of this study, the domain related to MA was reclassified from critical to noncritical, as SRs with and without MA were included. The other six critical domains were used according to the tool guidelines. 18 Two reviewers (PV and AP) independently responded to each of the 16 items in the checklist with either "yes," "partial yes," or "no." Based on the interpretations of the critical and noncritical domains, the overall quality of the review was rated as high, moderate, low, or critically low. 18 Disagreements were resolved through discussion or by consulting a third reviewer.

Data synthesis
To provide an understandable summary of existing evidence syntheses, characteristics of the methods evaluated in the included SRs were examined and key findings were categorized and presented based on the corresponding step in the SR process. The categories of key elements within each step were discussed and agreed by the authors. Results of the included reviews were tabulated and summarized descriptively, along with a discussion on any overlap in the primary studies. 15 No quantitative analyses of the data were performed.

Characteristics of included reviews
SRs evaluated methods used in five different steps in the SR process, including methods used in defining the scope of review (n = 3), literature search (n = 3), study selection (n = 2), data extraction (n = 1), and RoB assessment (n = 2) ( Table 2).
There was some overlap in the primary studies evaluated in the included SRs on the same topics: Schmucker et al. 26

Methodological quality of included reviews
Overall, the quality of the included reviews was assessed as moderate at best ( Table 2). The most common critical weakness in the reviews was failure to provide justification for excluding individual studies (four reviews). Detailed quality assessment is provided in Appendix C.

Methods for defining review scope and eligibility
Two SRs investigated the effect of excluding data obtained from gray or unpublished sources on the pooled effect estimates of MA. 21,26 Hopewell et al. 21

Quality of review
Step: Defining the review scope and eligibility Excluding study data based on publication status  In an SR comparing handsearching and electronic database searching, Hopewell et al. 20 found that handsearching retrieved more relevant RCTs (retrieval rate of 92%−100%) than searching in a single electronic database (retrieval rates of 67% for PsycINFO/PsycLIT, 55% for MEDLINE, and 49% for Embase). The retrieval rates varied depending on the quality of handsearching, type of electronic search strategy used (e.g., simple, complex or CHSSS), and type of trial reports searched (e.g., full reports, conference abstracts, etc.). The authors concluded that handsearching was particularly important in identifying full trials published in nonindexed journals and in languages other than English, as well as those published as abstracts and letters. 20 The effectiveness of checking reference lists to retrieve additional relevant studies for an SR was investigated by Horsley et al. 22 The review reported that checking reference lists yielded 2.5%-40% more studies depending on the quality and comprehensiveness of the electronic search used. The authors conclude that there is some evidence, although from poor quality studies, to support use of checking reference lists to supplement database searching. 22

Methods for selecting studies
Three approaches relevant to reviewer characteristics, including number, experience, and blinding of reviewers involved in the screening process were highlighted in an SR by Robson et al. 14 Based on the retrieved evidence, the authors recommended that two independent, experienced, and unblinded reviewers be involved in study selection. 14 A modified approach has also been suggested by the review authors, where one reviewer screens and the other reviewer verifies the list of excluded studies, when the resources are limited. It should be noted however this suggestion is likely based on the authors' opinion, as there was no evidence related to this from the studies included in the review.
Robson et al. 14 also reported two methods describing the use of technology for screening studies: use of Google Translate for translating languages (for example, German language articles to English) to facilitate screening was considered a viable method, while using two computer monitors for screening did not increase the screening efficiency in SR. Title-first screening was found to be more efficient than simultaneous screening of titles and abstracts, although the gain in time with the former method was lesser than the latter. Therefore, considering that the search results are routinely exported as titles and abstracts, Robson et al. 14 recommend screening titles and abstracts simultaneously. However, the authors note that these conclusions were based on very limited number (in most instances one study per method) of low-quality studies. 14

Methods for data extraction
Robson et al. 14 examined three approaches for data extraction relevant to reviewer characteristics, including number, experience, and blinding of reviewers (similar to the study selection step). Although based on limited evidence from a small number of studies, the authors recommended use of two experienced and unblinded reviewers for data extraction. The experience of the reviewers was suggested to be especially important when extracting continuous outcomes (or quantitative) data. However, when the resources are limited, data extraction by one reviewer and a verification of the outcomes data by a second reviewer was recommended.
As for the methods involving use of technology, Robson et al. 14 identified limited evidence on the use of two monitors to improve the data extraction efficiency and computer-assisted programs for graphical data extraction. However, use of Google Translate for data extraction in non-English articles was not considered to be viable. 14 28 Further studies are required to examine this aspect.
Although we acknowledge the lack of standardized quality assessment tools for methodological study designs, we adhered to the Cochrane criteria for identifying SRs in this overview. This was done to ensure consistency in the quality of the included evidence. As a result, we excluded three reviews that did not provide any form of discussion on the quality of the included studies. The methods investigated in these reviews concern supplementary search, 29 data extraction, 12 and screening. 13 However, methods reported in two of these three reviews, by Mathes et al. 12 and Waffenschmidt et al., 13 have also been examined in the SR by Robson et al., 14 which was included in this overview; in most instances (with the exception of one study included in Mathes et al. 12 and Waffenschmidt et al. 13 each), the studies examined in these excluded reviews overlapped with those in the SR by Robson et al. 14 One of the key gaps in the knowledge observed in this overview was the dearth of SRs on the methods used in the data synthesis component of SR. Narrative and quantitative syntheses are the two most commonly used approaches for synthesizing data in evidence synthesis. 5 There are some published studies on the proposed indications and implications of these two approaches. 30,31 These studies found that both data synthesis methods produced comparable results and have their own advantages, suggesting that the choice of the method must be based on the purpose of the review. 31 With increasing number of "expedited" SR approaches (so called "rapid reviews") avoiding MA, 10,11 further research studies are warranted in this area to determine the impact of the type of data synthesis on the results of the SR.

Implications for future research
The findings of this overview highlight several areas of paucity in pri-

Strengths and limitations
To our knowledge, this is the first overview summarizing current evidence from SRs and MA on different methodological approaches used in several fundamental steps in SR conduct. The overview methodology followed well established guidelines and strict criteria defined for the inclusion of SRs.
There are several limitations related to the nature of the included reviews. Evidence for most of the methods investigated in the included reviews was derived from a limited number of primary studies. Also, the majority of the included SRs may be considered outdated as they were published (or last updated) more than 5 years ago 33 ; only three of the nine SRs have been published in the last 5 years. 14,25,26 Therefore, important and recent evidence related to these topics may not have been included. Substantial numbers of included SRs were conducted in the field of health, which may limit the generalizability of the findings.
Some method evaluations in the included SRs focused on quantitative analyses components and MA conclusions only. As such, the applicability of these findings to SR more broadly is still unclear. 28 Considering the methodological nature of our overview, limiting the inclusion of SRs according to the Cochrane criteria might have resulted in missing some relevant evidence from those reviews without a quality assessment component. 12,13,29 Although the included SRs performed some

CONCLUSIONS
This overview identified limited SR-level evidence on various methodological approaches currently employed during five of the seven fundamental steps in the SR process. Limited evidence was also identified on some methodological modifications currently used to expedite the SR process. Overall, findings highlight the dearth of SRs on SR methodologies, warranting further work to confirm several current recommendations on conventional and expedited SR processes.

ACKNOWLEDGMENTS
The first author is supported by a La Trobe University Full Fee Research Scholarship and a Graduate Research Scholarship.
Open Access Funding provided by La Trobe University.